Lingual thyroid is a rare embryological anomaly originated from the thyroid gland failure that descends from the foramen cecum to its normal eutopic pre-laryngeal site. The case in this study was a 39. year old female, presenting with the sensation of a foreign body, progressive dysphagia and dyspnea. Indirect laryngoscopy revealed a large well-defined mass in the tongue base. Imaging studies confirmed the diagnosis of large ectopic lingual thyroid. The surgery was performed via an external cervical approach due to the mass size. The decision on the best treatment looks into the mass position, size, symptoms, airway emergency and medical facilities. Â© 2014

Bayazian, G.

Ghiasi, S.

Khansarinia, A.

English

eprints Iran University of Medical Sciences

Egyptian Journal of Ear, Nose, Throat and Allied Sciences (2015) 16, 95–99HO ST E D  BYEgyptian Society of Ear, Nose, Throat and Allied SciencesEgyptian Journal of Ear, Nose, Throat and AlliedScienceswww.ejentas.comREVIEWDysphagia and dyspnea by lingual thyroidmass: An appropriate approach* Corresponding author. Tel.: +98 9126953782; fax: +982166511011.E-mail addresses: bayazian.g@iums.ac.ir, gr_bayazian@yahoo.com(G. Bayazian).Peer review under responsibility of Egyptian Society of Ear, Nose,Throat and Allied Sciences.http://dx.doi.org/10.1016/j.ejenta.2014.11.0022090-0740 ª 2014 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by Elsevier B.V. All rights reservSamad Ghiasi a, Gholamreza Bayazian b,*, Arezoo Khansarinia ca Department of Otolaryngology, Tabriz University of Medical Sciences, Iranb Department & Research Center of Otolaryngology, Hazrat Rasoul Akram Hospital, Iran University of Medical Sciences,Tehran, Iranc Tabriz University of Medical Sciences, IranReceived 8 June 2014; accepted 20 November 2014Available online 8 December 2014KEYWORDSEctopic thyroid;Lingual thyroid;Thyroid dysgenesis;Dysphagia;DyspneaAbstract Lingual thyroid is a rare embryological anomaly originated from the thyroid gland fail-ure that descends from the foramen cecum to its normal eutopic pre-laryngeal site. The case in thisstudy was a 39 year old female, presenting with the sensation of a foreign body, progressive dyspha-gia and dyspnea. Indirect laryngoscopy revealed a large well-defined mass in the tongue base. Imag-ing studies confirmed the diagnosis of large ectopic lingual thyroid. The surgery was performed viaan external cervical approach due to the mass size. The decision on the best treatment looks into themass position, size, symptoms, airway emergency and medical facilities.ª 2014 Egyptian Society of Ear, Nose, Throat and Allied Sciences. Production and hosting by ElsevierB.V. All rights reserved.Contents1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 962. Case. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 963. Surgical technique . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 964. Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 965. Conclusion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 98Conflict of interest . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 98References. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 98ed.Figure 1 CTscan with contrast revealed a large enhanced massat tongue base.96 S. Ghiasi et al.1. IntroductionLingual thyroid is a rare developmental anomaly that origi-nates from aberrant embryogenesis during the passage of thethyroid gland through the neck. Embryonic developmentbegins around 24 days after fertilization in the floor of thePrimitive hypopharynx out of the median endodermal thicken-ing. The primitive gland starts to descend close to the hyoidbone and the laryngeal cartilages.1The thyroglossal duct is a narrow tube which connects thedeveloping thyroid gland to the tongue and it usually involutesduring weeks 6–8. The foramen cecum is the opening of thethyroglossal duct in the tongue. The thyroid gland graduallydescends to meet the lateral ultimo-branchial bodies; thefusion of these elements forms the functional and mature thy-roid gland by the third fetal month.2Lingual thyroid is the most frequent ectopic location for thethyroid gland, although its prevalence varies between 1:100,000and 1:300,000. The clinical incidence is reported to range from1:4000 to 1:10,000. Its true incidence is probably underesti-mated; some authors reported the ectopic lingual tissue 10%out of 200 consecutive necropsies.3 In literature, approximately400 symptomatic cases were presented.4 Ectopic thyroid tissuecan also occur between the geniohyoid and mylohyoid muscles(sublingual thyroid), above the hyoid bone (pre-laryngeal thy-roid) or in such rare sites as the mediastinum, precardial sac,heart, breasts, pharynx, esophagus, trachea, lungs, duodenum,mesentery of the small intestine, and adrenal gland.5–9Most ectopic thyroids are asymptomatic; instead, patientscomplain from some upper airway problems such as foreignbody sensation, progressive dysphagia to solid foods, odyno-phagia, hoarseness, hot potato voice, bleeding and dyspnea.Some patients have difficulty in breathing or snoring on theirback position. Although symptoms grow very gradually, somepatients may experience acute onset of obstructive airwaysymptoms.2. CaseA 39-year-old female was admitted with dyspnea and mufflevoice by the emergency ward at Imam Reza Hospital, Tabriz,Iran. The case had a mass in the tongue base with a foreignbody sensation and stomatolalia that caused dyspnea and dys-phagia to solid food. The patient was unable to lie on her backfor last 5 years. The airway obstruction caused mouth breath-ing and snoring. These symptoms showed gradual growing inthe last 10 years. Weight loss was another unexpected symp-tom. No record for thyroid disease was available except anuntreated mild depression.In the oral examination, the patient had a solid, pink andspherical mass, covered with intact mucosa on the base ofthe tongue, obstructing the visualization of the larynx. Neckexamination revealed neither palpable thyroid glands in thenormal pre-tracheal position, nor cervical adenopathies. Thecomputed tomography (CT) revealed a 41 · 34 · 45 mm masswith distinct margins restricted to the tongue base enhancingafter contrast administration. Absence of the thyroid glandswas notable (Fig. 1).A thyroid scan with technetium Tc-99m sodium showed amarked isotope uptake in the area of the tongue base withno uptake in the neck (Fig. 2). Tests of thyroid hormonesshowed normal thyroid-stimulating hormone (TSH) concen-trations and normal Free T3 and Free T4.3. Surgical techniqueIn this case, an external approach with transhyoid medianpharyngotomy was utilized. The anesthesiologist was unableto intubate the patient even with glidescope due to the largemass kissing the posterior pharynx wall. Although awakefiberoptic nasotracheal intubation is reported as the commonoption in these patients, the tracheotomy under local anesthe-sia was initially done due to the insufficient equipment. It wasfollowed by the operation under general anesthesia.An 8 cm transverse midline incision was done in the neckunder the hyoid bone. The hyoid bone and the suprahyoidmuscles were exposed, the infra-hyoid and supra-hyoid mus-cles were freed and the hyoid bone body was removed.Another incision was done in the pharyngeal mucosa to enterthe hypopharynx. The mass visualized at the tongue base wasdissected with an under mucosa approach and excised (Figs. 3and 4). The pharyngeal opening was sutured in 2 layers, andthe neck incision was closed in 3 layers. A nasogastric tubewas introduced to feed the patient for 10 days. The tracheos-tomy tube was removed after 5 days. On the 8th post-operativeday, the patient was discharged. The substitutive hormonetherapy was commenced to maintain the euthyroid state. Theexcised specimen was sent for microscopic examination whichwas reported as a normal thyroid tissue.4. DiscussionA rare embryological aberration, ectopic thyroid may occurwhen the migration of the thyroid results in the lingual (at ton-gue base), sublingual (below the tongue), prelaryngeal (in frontof the larynx), and substernal (in the mediastinum) ectopy.Dual ectopic thyroid is described1,10 even with the thyroidglands in the normal location.11 It is more frequent in femaleswith the ratio 4:1 to men. Ectopic thyroid is seen at any age butmore commonly during childhood, adolescence or aroundmenopause. This anomaly probably occurs when the demandFigure 2 Scan with Tc-99m showed massive uptake at tongue base but no uptake at neck.Figure 3 The mass was dissected within pharynx throughtranshyoid approach.Figure 4 Large well defined thyroid mass was removed.Dysphagia and dyspnea by lingual thyroid mass 97for thyroid hormones increases while it will increase the circu-lating TSH levels with the growth of ectopic thyroid tissues.12About 33–62% of the patients with ectopic thyroid showhypothyroidism with increased level of TSH.5,13 Most ectopicthyroids are asymptomatic with no required therapies. Symp-toms are related to the growth of the thyroid tissue, causingdysphagia, dysphonia, stomatolalia, bleeding or dyspnea.4–14Acute airway compromise in these patients is very rare in liter-ature, although it is possible because of acute bleeding in theectopic thyroid. Management in this emergency situationdepends on available facilities. Fiberoptic-assisted intubationseems the best but temporary tracheostomy and positive pres-sure mask ventilation are other alternatives. The enlarged lin-gual thyroid has been described as a cause of difficultintubation during induction of anesthesia, especially in caseof bleeding.10,14Clinically, lingual thyroid presents itself as a pink and firmmass at the tongue base. The most important diagnostic tool isthe thyroid scan with technetium Tc-99m sodium. Ultrasonog-raphy, CT and magnetic resonance imaging (MRI), however,may help to define the extension and location of the ectopicthyroid gland. Thyroid scan can also reveal whether thereare other sites of thyroid tissues. In approximately 75% ofthe patients, the ectopic tissue is the only functioning thyroidtissue in the body.15 Therefore, it is important to follow upthe patients for the risk of post-operative hypothyroidism.Differential diagnosis for tongue base mass includes lym-phangioma, minor salivary gland tumors, midline bronchialcysts, thyroglossal duct cysts, epidermal and sebaceous cysts,angioma, adenoma, fibroma and lipomas.5,10Managing lingual thyroid is still controversial. There is noconsensus about the optimal therapeutic strategy, perhaps due98 S. Ghiasi et al.to the rarity of this clinical entity.16 No treatment is requiredwhen the lingual thyroid is asymptomatic and the patient isin the euthyroid state. Careful observations are recommendedin order to trace any developing complications. Malignanttransformation has been reported in a few cases.17Some researchers consider the complete surgical removal ofthe thyroid gland as an appropriate treatment.18–21 Forpatients with no or mild clinical symptoms and elevatedTSH concentration, the substitutive therapies with thyroidhormone might be successful to gradually reduce the mass size.Ablative radioiodine therapy is another alternative approachrecommended in aged patients or patients unfit for surgery.This treatment should be avoided in children and young adultsas the required systemic dosage has some potentially damagingeffects on the gonads or other organs. Moreover, the thyroidtissues are often hypoactive and the required dosage of radio-iodine is generally high.22Surgery is the best option when medical therapies fail insymptomatic or complicated cases. In literature, several surgi-cal approaches are described such as surgical removal of themass with external approaches, trans-hyoid, lateral pharyngot-omy, or trans-oral ablation of the mass. The trans-oralapproaches seem to prevent injuries to deep neck structuresand other possible complications (injuries to the lingual nerve,fistula formation, deep cervical infection and visible scars) andcould be considered as a significant treatment for small masses.Since the external approaches with temporary tracheotomyprevent bleeding, they are recommended in the case of bulkymasses.For trans-oral approaches, cold instruments with monopo-lar coagulation and laser CO2 are known as practical.14,23 Newtechnique has been recently used for surgery is TransoralRobotic Surgery.24 Although authors suggested this methodas a feasible and safe alternative to other invasive surgicalapproaches in patients with symptomatic lingual thyroid, butexperience with this technique is so low and the equipmentmay be unavailable anywhere.The possibility that the ectopic thyroid tissues could be theonly functioning tissues must be carefully considered. By someresearchers, transplantation of the excised ectopic tissue is rec-ommended to avoid permanent hypothyroidism.4 As an alter-native, substitutive hormone treatment may start to preservethe euthyroid states. Transplantation is not necessary in thecase of partial surgical eradication since the substitutive ther-apy seems necessary to preserve euthyroid state and avoidrecurrence of the mass.Based on the results in this case study, the researchersbelieve that the choice of the appropriate treatment is some-how problematic considering patients’ general conditions, thelesion size and the presence of local symptoms or complica-tions such as hemorrhage, cystic degeneration or malignan-cies.18–21 In symptomatic, old patients, unfit to undergomore aggressive surgical approaches, or in small and anteriorlesions, the partial trans-oral approaches are recommended.The partial trans-oral approach is less aggressive but cannotprevent malignant transformation and possible relapses.Moreover, it allows less control of bleeding during the surgery.Total ablation is considered as the most appropriateapproach in younger patients, in the case of large lesions ordeeply located in the caudal part of the tongue base. In suchcases, transplantation of the thyroid tissue is not necessarysince substitutive hormone treatment is highly recommendedas a radical surgical approach for normal thyroid gland.25Special care should be taken to ensure the patient’s airwaypatency before and after surgery. The major risk to thesepatients is that insertion of any object into the oral cavitycould traumatize the glandular tissue causing subsequent hem-orrhage and a compromised airway. This could occur with asimple oropharyngeal airway or during laryngoscopy. Someauthors suggested procedures requiring general anesthesia inthese patients should be carried out using fiberoptic-assistednasal intubation.26 Temporary tracheostomy is another choiceif fiberoptic-assisted intubation was not available as in ourcase, she underwent temporary tracheostomy for 5 days. Aftersurgery also maintaining the airway is critical and it can usu-ally be achieved by temporary tracheostomy or maintainingthe naso-tracheal tube for the first hours after surgery. Trache-ostomy or intubation should not be removed until hemostasisand airway patency have been confirmed.255. ConclusionThe critical point in diagnosing lingual thyroid is consideringthis entity. In the case of patients complaining from sphericalmasses, conducting indirect laryngoscopy and observing thetongue base may raise clinical suspicion while the accessibleimaging options may confirm diagnosis. The patients are oftenadmitted in the emergency ward with a sudden increase in themass size, asthma and dyspnea; hence, the lingual thyroid is apossibility.Most researchers believe that the best treatment is deter-mined by the mass position, size, symptoms, airway emergency,medical facilities such as laser therapy, and last but not the leastthe experience of the surgeon. The operated patients need a fol-low-up treatment for side effects or hormone substitution.Conflict of interestNone.References1. Ulug T, Ulubil SA, Alagol F. Dual ectopic thyroid: report of a case.J Laryngol Otol. 2003;117:574–576.2. Chanin LR, Greenberg LM. Pediatric upper airway obstruction dueto ectopic thyroid: classification and case reports. Laryngoscope.1988;98:422–427.3. Sauk JJ. Ectopic lingual thyroid. J Pathol. 1970;102:239–243.4. Gallo A, Leonetti F, Torri E, Manciocco V, Simonelli M,DeVincentiis M. Ectopic lingual thyroid as unusual cause of severedysphagia. Dysphagia. 2001;16:220–223.5. Di Benedetto V. Ectopic thyroid gland in the submandibular regionsimulating a thyroglossal duct cyst: a case report. J Pediatr Surg.1997;32:1745–1746.6. Kumar R, Sharma S, Marwah A, Moorthy D, Dhanwal D,Malhotra A. Ectopic goiter masquerading as submandibular glandswelling: a case report and review of the literature. Clin Nucl Med.2001;26:306–309.7. Pollice L, Caruso G. Struma cordis. Ectopic thyroid goiter in theright ventricle. Arch Pathol Lab Med. 1986;110:452–453.8. Porqueddu M, Antona C, Polvani G, et al. Ectopic thyroid tissuein the ventricular outflow tract: embryologic implications. Cardi-ology. 1995;86:524–526.Dysphagia and dyspnea by lingual thyroid mass 999. Ferlito A, Giarelli L, Silvestri F. Intratracheal thyroid. J LaryngolOtol. 1988;102:95–96.10. Hazarika P, Siddiqui SA, Pujary K, Shah P, Nayak DR,Balakrishnan R. Dual ectopic thyroid: a report of two cases. JLaryngol Otol. 1998;112:393–395.11. Huang TS, Chen HY. Dual thyroid ectopia with a normallylocated pretracheal thyroid gland: case report and literaturereview. Head Neck. 2007;29:885–888.12. Steinwald Jr OP, Muehrcke RC, Economou SG. Surgical correc-tion of complete lingual ectopia of the thyroid gland. Surg ClinNorth Am. 1970;50:1177–1186.13. Yoon JS, Won KC, Cho IH, Lee JT, Lee HW. Clinicalcharacteristics of ectopic thyroid in Korea. Thyroid.2007;17:1117–1121.14. Hafidh MA, Sheahan P, Khan NA, Colreavy M, Timon C. Roleof CO2 laser in the management of obstructive ectopic lingualthyroids. J Laryngol Otol. 2004;118:807–809.15. Baik SH, Choi JH, Lee HM. Dual ectopic thyroid. Eur ArchOtorhinolaryngol. 2002;259:105–107.16. Noussios G, Anagnostis P, Goulis DG, Lappas D, Natsis K.Ectopic thyroid tissue: anatomical, clinical, and surgical implica-tions of a rare entity. Eur J Endocrinol. 2011;165(3):375–382.17. Shah BC, Ravichand CS, Juluri S, Agarwal A, Pramesh CS,Mistry RC. Ectopic thyroid cancer. Ann Thorac Cardiovasc Surg.2007;13:122–124.18. Paludetti G, Galli J, Almadori G, Ottaviani F, d’Alatri L, MauriziM. Ectopic thyroid gland. Acta Otorhinolaryngol Ital.1991;11:117–133.19. Amr B, Monib S. Lingual thyroid: a case report. Int J Surg CaseRep. 2011;2(8):313–315.20. Goldstein B, Westra WH, Califano J. Multifocal papillary thyroidcarcinoma arising in a lingual thyroid: a case report. ArchOtolaryngol Head Neck Surg. 2002;128:1198–1200.21. Galizia G, Lieto E, Ferrara A, et al. Ectopic thyroid: report of acase. G Chir. 2001;22:85–88.22. Weider DJ, Parker W. Lingual thyroid: review, case reports, andtherapeutic guidelines. Ann Otol Rhinol Laryngol.1977;86:841–848.23. Puxeddu R, Pelagatti CL, Nicolai P. Lingual thyroid: endoscopicmanagement with CO2 laser. Am J Otolaryngol. 1998;19:136–139.24. Park YM, Kim WS, Byeon HK, Lee SY, Kim SH. A noveltechnique for the resection of the symptomatic lingual thyroid:transoral robotic surgery. Thyroid. 2013;23(4):466–471.25. Toso A, Colombani F, Averono G, Aluffi P, Pia F. Lingualthyroid causing dysphagia and dyspnoea. Case reports and reviewof the literature. Acta Otorhinolaryngol Ital. 2009;29(4):213.26. Buckland RW, Pedley J. Lingual thyroid – a threat to the airway.Anaesthesia. 2000;55(11):1103–1105.

Dysphagia and dyspnea by lingual thyroid mass: An appropriate approach

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Dysphagia and dyspnea by lingual thyroid mass: An appropriate approach

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